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The epidermis was ulcerated&#44; and the lateral acanthotic epidermis extended downwards in a tongue-like projection &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; Tumor cells were composed of different types of cells&#44; such as eosinophilic&#44; basophilic&#44; and transitional cells&#46; Partial elimination of tumor cells through the epidermis was observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; There was an inflammatory reaction with mononuclear cells&#44; and a number of foreign-body type giant cells were found around the mass&#46; We retrospectively examined three specimens of previously resected tumors&#46; One of the specimens also showed proliferation of eosinophilic and basaloid tumor cells in the dermis with central ulceration of the epidermis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>c&#41;&#44; whereas the other two tumors were pilomatricoma without perforation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Perforating pilomatricoma is a rare variant of pilomatricoma&#44; which clinically presents with inflammatory papules&#44; cutaneous horn-like nodules&#44; craters&#44; and ulcers resembling keratoacanthoma or solid masses&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;4</span></a> Most of the reported cases are solitary lesions&#44; and one case has been reported in which perforating pilomatricoma was observed in one of the multiple pilomatricomas&#59;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> however&#44; to our knowledge&#44; only one case of multiple onset of perforating pilomatricomas has been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In that case&#44; the patient presented Churg-Strauss syndrome and Rubinstein-Taybi syndrome and developed a total of 15 tumors&#46; Although the exact number was not specified&#44; multiple lesions were classified as perforating pilomatricomas&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our patient was suspected of myotonic dystrophy&#44; which&#44; however&#44; was not genetically confirmed&#46; Myotonic dystrophy is an autosomal dominant inherited disease caused by a mutation in the dystrophia myotonica protein kinase gene on chromosome 19&#46; The occurrence of multiple pilomatricomas has been reported to be associated with myotonic dystrophy&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Our patient developed multiple firm nodules on the nape&#44; back&#44; and upper arm&#46; The total number of the nodules was five&#44; four of which were subjected to histopathological examination&#46; All of the four nodules were diagnosed as pilomatricomas&#46; Of interest&#44; two of the nodules were perforating pilomatricomas&#44; showing perforation and partial elimination of the eosinophilic tumor cells&#46; Recent studies have shown that matrix metalloproteinase-9 and -12 derived from tumor cells&#44; fibroblasts&#44; and macrophages may be relevant to the partial elimination of the tumor&#44; by degradation of collagen and elastic fibers&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> When superficially located&#44; the elimination of pilomatricoma is accelerated by virtue of epidermal catharsis mechanisms&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0015" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#8217; contributions</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mai Endo&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; wrote the initial draft of the manuscript&#59; read and approved the final version of the manuscript&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Toshiyuki Yamamoto&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; assisted in the preparation of the manuscript&#59; read and approved the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Case Letter
A case of multiple perforating pilomatricomas
Mai Endo
Corresponding author
enmai04@fmu.ac.jp

Corresponding author.
, Toshiyuki Yamamoto
Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 22-year-old man was referred to our department for evaluation of a slightly painful nodule on the arm that had increased in size over the previous four months&#46; He had a history of surgical resection of several nodules on the nape&#44; back&#44; and arm at other dermatology clinics&#46; He complained of myotonic symptoms with difficulty in relaxing his hands after contraction and was followed at the Neurology Department of our hospital under suspicion of myotonic dystrophy&#44; but he refused to undergo genetic testing&#46; Physical examination showed a 10&#8239;&#215;&#8239;10&#8239;mm&#44; slightly elevated&#44; firm reddish nodule with central brownish crusts on the left upper arm &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>a&#41;&#46; The tumor was surgically removed under local anesthesia&#46; Histopathological examination showed a well-circumscribed tumor mass penetrating the dermis&#46; The epidermis was ulcerated&#44; and the lateral acanthotic epidermis extended downwards in a tongue-like projection &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; Tumor cells were composed of different types of cells&#44; such as eosinophilic&#44; basophilic&#44; and transitional cells&#46; Partial elimination of tumor cells through the epidermis was observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; There was an inflammatory reaction with mononuclear cells&#44; and a number of foreign-body type giant cells were found around the mass&#46; We retrospectively examined three specimens of previously resected tumors&#46; One of the specimens also showed proliferation of eosinophilic and basaloid tumor cells in the dermis with central ulceration of the epidermis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>c&#41;&#44; whereas the other two tumors were pilomatricoma without perforation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Perforating pilomatricoma is a rare variant of pilomatricoma&#44; which clinically presents with inflammatory papules&#44; cutaneous horn-like nodules&#44; craters&#44; and ulcers resembling keratoacanthoma or solid masses&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;4</span></a> Most of the reported cases are solitary lesions&#44; and one case has been reported in which perforating pilomatricoma was observed in one of the multiple pilomatricomas&#59;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> however&#44; to our knowledge&#44; only one case of multiple onset of perforating pilomatricomas has been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In that case&#44; the patient presented Churg-Strauss syndrome and Rubinstein-Taybi syndrome and developed a total of 15 tumors&#46; Although the exact number was not specified&#44; multiple lesions were classified as perforating pilomatricomas&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our patient was suspected of myotonic dystrophy&#44; which&#44; however&#44; was not genetically confirmed&#46; Myotonic dystrophy is an autosomal dominant inherited disease caused by a mutation in the dystrophia myotonica protein kinase gene on chromosome 19&#46; The occurrence of multiple pilomatricomas has been reported to be associated with myotonic dystrophy&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Our patient developed multiple firm nodules on the nape&#44; back&#44; and upper arm&#46; The total number of the nodules was five&#44; four of which were subjected to histopathological examination&#46; All of the four nodules were diagnosed as pilomatricomas&#46; Of interest&#44; two of the nodules were perforating pilomatricomas&#44; showing perforation and partial elimination of the eosinophilic tumor cells&#46; Recent studies have shown that matrix metalloproteinase-9 and -12 derived from tumor cells&#44; fibroblasts&#44; and macrophages may be relevant to the partial elimination of the tumor&#44; by degradation of collagen and elastic fibers&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> When superficially located&#44; the elimination of pilomatricoma is accelerated by virtue of epidermal catharsis mechanisms&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0015" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#8217; contributions</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mai Endo&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; wrote the initial draft of the manuscript&#59; read and approved the final version of the manuscript&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Toshiyuki Yamamoto&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; assisted in the preparation of the manuscript&#59; read and approved the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Article information
ISSN: 03650596
Original language: English
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Idiomas
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